Literature DB >> 29427788

Split hand-foot malformation and a novel WNT10B mutation.

Piranit Nik Kantaputra1, Seema Kapoor2, Prashant Verma2, Worrachet Intachai3, James R Ketudat Cairns4.   

Abstract

We report an Indian girl with split-hand/foot malformation (SHFM), sparse hair, and interrupted eyebrows, who carries a novel homozygous deletion c.695_697delACA in WNT10B. The variant is deduced to cause an in-frame deletion of Asn residue 232 (p.Asn232del). According to the protein model, this single amino acid deletion at the critical position in the protein structure is likely to severely affect the protein structure and function. This deletion is likely to lead decreased lifetime and make it unable to bind to its receptors and other ligands. The patient and all family members had normal bone density and they were not obese like some of the patients with WNT10B variants. Here we report a patient with SHFM6 who carried a novel WNT10B mutation. Sparse hair and interrupted eyebrows may be associated findings of SHFM6.
Copyright © 2018 Elsevier Masson SAS. All rights reserved.

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Year:  2018        PMID: 29427788     DOI: 10.1016/j.ejmg.2018.02.001

Source DB:  PubMed          Journal:  Eur J Med Genet        ISSN: 1769-7212            Impact factor:   2.708


  4 in total

1.  WNT1-associated osteogenesis imperfecta with atrophic frontal lobes and arachnoid cysts.

Authors:  Piranit Nik Kantaputra; Yuddhasert Sirirungruangsarn; Pannee Visrutaratna; Sasitorn Petcharunpaisan; Bruce M Carlson; Worrachet Intachai; Jutamas Sudasna; Jatupol Kampuansai; Prapai Dejkhamron
Journal:  J Hum Genet       Date:  2019-01-28       Impact factor: 3.172

2.  Developmental genomics of limb malformations: Allelic series in association with gene dosage effects contribute to the clinical variability.

Authors:  Ruizhi Duan; Hadia Hijazi; Elif Yilmaz Gulec; Hatice Koçak Eker; Silvia R Costa; Yavuz Sahin; Zeynep Ocak; Sedat Isikay; Ozge Ozalp; Sevcan Bozdogan; Huseyin Aslan; Nursel Elcioglu; Débora R Bertola; Alper Gezdirici; Haowei Du; Jawid M Fatih; Christopher M Grochowski; Gulsen Akay; Shalini N Jhangiani; Ender Karaca; Shen Gu; Zeynep Coban-Akdemir; Jennifer E Posey; Yavuz Bayram; V Reid Sutton; Claudia M B Carvalho; Davut Pehlivan; Richard A Gibbs; James R Lupski
Journal:  HGG Adv       Date:  2022-08-04

Review 3.  Signaling Pathways in Bone Development and Their Related Skeletal Dysplasia.

Authors:  Alessandra Guasto; Valérie Cormier-Daire
Journal:  Int J Mol Sci       Date:  2021-04-21       Impact factor: 5.923

Review 4.  WNT Signaling and Bone: Lessons From Skeletal Dysplasias and Disorders.

Authors:  Yentl Huybrechts; Geert Mortier; Eveline Boudin; Wim Van Hul
Journal:  Front Endocrinol (Lausanne)       Date:  2020-04-09       Impact factor: 5.555

  4 in total

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